GBE1-related disorders: Adult polyglucosan body disease and its neuromuscular phenotypes.
Paulo Victor Sgobbi de SouzaBruno Mattos Lombardi BadiaIgor Braga FariasWladimir Bocca Vieira de Rezende PintoAcary Souza Bulle OliveiraHasan Orhan AkmanSalvatore DiMauroPublished in: Journal of inherited metabolic disease (2020)
Adult polyglucosan body disease (APBD) represents a complex autosomal recessive inherited neurometabolic disorder due to homozygous or compound heterozygous pathogenic variants in GBE1 gene, resulting in deficiency of glycogen-branching enzyme and secondary storage of glycogen in the form of polyglucosan bodies, involving the skeletal muscle, diaphragm, peripheral nerve (including autonomic fibers), brain white matter, spinal cord, nerve roots, cerebellum, brainstem and to a lesser extent heart, lung, kidney, and liver cells. The diversity of new clinical presentations regarding neuromuscular involvement is astonishing and transformed APBD in a key differential diagnosis of completely different clinical conditions, including axonal and demyelinating sensorimotor polyneuropathy, progressive spastic paraparesis, motor neuronopathy presentations, autonomic disturbances, leukodystrophies or even pure myopathic involvement with limb-girdle pattern of weakness. This review article aims to summarize the main clinical, biochemical, genetic, and diagnostic aspects regarding APBD with special focus on neuromuscular presentations.
Keyphrases
- peripheral nerve
- white matter
- spinal cord
- skeletal muscle
- copy number
- induced apoptosis
- multiple sclerosis
- heart rate variability
- type diabetes
- heart rate
- muscular dystrophy
- metabolic syndrome
- cell death
- cell proliferation
- intensive care unit
- dna methylation
- oxidative stress
- adipose tissue
- endoplasmic reticulum stress